Archives of Epilepsy Case Report Sleep- Related Hypermotor Epilepsy: A Rare Case Leyla Köse Leba , Safiye Gül Kenar , Bülent Oğuz Genç Department of Neurology, Necmettin Erbakan University, Meram Medical Faculty, Konya, Turkey Cite this article as: Leba LK, Kenar SG, Genç BO. Sleep-related hypermotor epilepsy: A rare case. Arch Epilepsy. 2022;28(4):169-171. Corresponding Author: Leyla Köse Leba, E-mail: leylakos_e@hotmail.com Received: August 2, 2022 Accepted: October 3, 2022 Publication Date: December 27, 2022 DOI: 10.5152/ArchEpilepsy.2022.222529 Abstract Sleep-related hypermotor epilepsy is a subgroup of sleep-related epilepsies and is very rare among focal epilepsies. It is a type of epilepsy that can be confused with the primary diseases of sleep and is diagnosed late. Here, we present a case diagnosed with electroencephalography (video-EEG) monitoring 13 years later. Keywords: Epilepsy, sleep-related hypermotor epilepsy, video-EEG INTRODUCTION When sleep-related epilepsy is mentioned, epilepsies in which all or more than 90% of seizures occur during sleep come to mind. Sleep-related hypermotor epilepsies are a subgroup of sleep-related epilepsies. Semiological features with evident hypermotor behaviors are very characteristic of seizures. Seizures in clusters for a short time are often seen during sleep at night. It may originate from the frontal lobe or, more rarely, from the extra frontal lobe. 1 Sleep-related hypermotor epilepsies are quite rare among focal epilepsies, and their prevalence is 1.8/100 000. 2 It peaks in childhood and adoles- cence and often occurs during the Non- Rapid Eye Movements (NREM) sleep phase and causes poor sleep quality. Sleep-associated hypermotor epilepsy is a late-diagnosed type of epilepsy and can be confused with primary sleep disorders. This article found it appropriate to present a case with recurrent sleep episodes for 13 years. She was followed up with the diagnosis of non-epileptic psychogenic seizures without treatment but was diagnosed correctly only with video-EEG monitoring. CASE PRESENTATION A 22-year-old female patient with a known diagnosis of epilepsy and using antiepileptic therapy presented to the clinic because of an increase in the frequency of seizures over the last 2 weeks. Involuntary movements started when she was nine, such as leaping in her hands and feet, often occur during falling asleep and waking up from sleep. A few years later, she had a generalized seizure accompanied by loss of consciousness, jaw locking, and convulsions throughout the body. Valproic acid was started with the diagnosis of epilepsy in the patient who had no pathology in EEG and magnetic resonance imaging. After 2-3 years of use, another physician said that she had psychogenic epilepsy and discontinued her treatment. During the drug-free period, the frequency of seizures was increased, and lamotrigine was started. She had seizures once a year or every two years for about 4-5 years under the lamotrigine treatment. However, recently, when she was stressed, her seizures started to be 1-2 times a week. The patient was hospitalized in the video-EEG unit for further examination and treatment; sharp wave discharges were noted in the bilateral parieto-occipital regions and were more prominent on the right in the interictal EEG (Figure 1A and B). She had 22 seizures, with a frequency of 5-8 per night, during the 4 days in the video-EEG monitorization. During these seizures, it was noted that she woke up suddenly from light sleep. She sometimes covered her mouth with her right hand and sometimes with both hands. Motor movements as pedaling both feet were accompanied by fearful facial expressions. It was observed that after these seizures in which awareness was lost, the patient recovered quickly and fell asleep again in a short time. When the patient was questioned retrospectively, she stated that she was unaware of what had happened. At the beginning of these attacks, which lasted between 30 and 70 seconds, rapid generalized activity was observed in the EEG, and epileptic activity appeared in the temporal region of the right hemisphere in the later periods of the seizure, mixed with rhythmic theta activity (Figure 1C and D). When we questioned the patient retrospectively, her attacks, which her relatives described as jumping, were similar to those seen during hospitalization and repeated almost every night. With the diagnosis of sleep-related hypermotor epilepsy, we planned to increase the dose of lamotrigine and switch to carbamazepine if it did not benefit when it reached the adequate dose. Content of this journal is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.